title: The challenge of measuring quality of life in children with Hirschsprung's disease or anorectal malformation.
abstract: OBJECTIVE
  The aim of the present study was to assess, after adaptation to French, the only specific quality of life (QoL) instrument for children with Hirschsprung's disease or anorectal malformation, the Hirschsprung's disease/Anorectal Malformation Quality of Life questionnaire (HAQL), in order to get a standardized QoL evaluation instrument that could further be used to help health care improvement.
  
  
  METHODS
  The study was conducted in three teaching hospitals, including the French reference center for anorectal and pelvic malformations. After adaptation to French, QoL questionnaires were sent to the children and proxies. The questionnaire was mailed to 280 families. Psychometrics properties of the questionnaires (validity and reliability) were analysed from 120 proxy and 96 child questionnaires.
  
  
  RESULTS
  The HAQL with the original structure was not acceptable. Exploratory steps led to a clinically pertinent structure that had acceptable fit and good validity and reliability properties. The final structure pools physical symptoms (continence, discomfort) and psychosocial dimensions (general well-being, social and emotional functioning) of QoL.
  
  
  CONCLUSIONS
  The final structure, despite the disadvantage of being a new structure, allows assessment of QoL in this population and has the advantage of being shorter and validated on the clinical postoperative questionnaire from the Krickenbeck international consensus.
authors.affiliation: Department of Pediatric surgery, University Hospital, Nantes, France; EA 4275 "Biostatistics, Pharmacoepidemiology and Subjective Measures in Health Sciences", Faculties of Medicine and Pharmaceutical Sciences, University of Nantes, Nantes, France. Electronic address: pclermidi@gmail.com.
authors.forename: Pauline
authors.initials: P
authors.lastname: Clermidi
authors.forename: Guillaume
authors.initials: G
authors.lastname: Podevin
authors.forename: Célia
authors.initials: C
authors.lastname: Crétolle
authors.forename: Sabine
authors.initials: S
authors.lastname: Sarnacki
authors.forename: Jean-Benoit
authors.initials: JB
authors.lastname: Hardouin
mesh_terms: D000071056:Anorectal Malformations; D001006:Anus, Imperforate / Q000150:complications / Q000523:psychology*; D002648:Child; D005602:France; D006305:Health Status Indicators; D006627:Hirschsprung Disease / Q000150:complications / Q000523:psychology*; D006801:Humans; D011581:Psychological Tests; D011594:Psychometrics; D011788:Quality of Life; D015203:Reproducibility of Results; D011795:Surveys and Questionnaires; D014175:Translating
keywords: Anorectal malformation; Disease-specific quality of life questionnaire; HAQL; Hirschsprung's disease; Quality of life